Cervicogenic exophthalmos: Possible etiology and pathogenesis

Background: Unilateral exophthalmos is often caused by inflammation, neoplasm, infection, metabolic disease, vascular disorder and several other less common conditions. Reflex sympathetic dystrophy related to unilateral exophthalmos has not been reported in the past literature.

Case summary: We describe a 45-year-old female with unilateral exophthalmos caused by reflex sympathetic dystrophy and its unexpected spontaneous disappearance after a standard anterior cervical discectomy and fixation operation with two PEEK interbody cages and a plate. To our surprise, the patient's left unilateral exophthalmos improved spontaneously in the morning on postoperative day 2-with no relapse, without any further medication, as of seven years. We have named this condition "cervicogenic exophthalmos."

Conclusion: We would inform other clinicians that unilateral exophthalmos was caused not only by inflammation, vascular disorder, infection, neoplasm, or metabolic disease, but also by reflex sympathetic dystrophy related with cervicogenic spondylosis. To the best of our knowledge, ours is the first related case report and use of the term "cervicogenic exophthalmos" after reviewing previous literature.